Osteoid humerus:
osteoma of the proximal Two misleading cases
Denls Katz, MD, and Her@ Thomazeau,
MD,
Ploemeur
and Rennes, France
Osteoid osteoma (00) is a common bone disease, making up 2.6% of all bone tumprs and 7 I % of all benign bone tumors. 2,6,8 The typical clinical features include variable pain, often worse at night and markedly responsive to salicylates, and a characteristic roentgenographic appearance of bony sclerosis around a lucent nidus. Although this might suggest an easy diagnosis, diagnosis can often be delayed I or 2 years, J4, 24 especial/y when the lesion is located near a joint.6, ” Such a lesion at the upper end of the humerus is rare. In a series of 245 cases DahW found only 5 cases out of 18 involving the humerus. Rockwood,2’ in a series grouping 00 and osteoblastoma together, identified a 10% to 15% frequency out of all shoulder tumors. The misleading aspects of juxfaarticular 00 are well knawn 2, 4.7, JO, 7 7, 75, 77, 19, 22, 23, 25 /j owever, involvement of the superior humeral epiphysis has only been reported briefly in one patient. I0 The two cases presented in this article show the diagnostic difficulfy encountered in this location and misleading clinical, radiologic, and sometimes histologic aspects of patient evaluation.
CASE 1 A 37-year-old woman had been treated for 2 years for pain in the right dominant shoulder. The initial diagnosis was calcifying tendinitis. The patient had received three steroid injections without any response and had undergone many sessions of physical therapy. Pain was variable and occurred day and night. Response to salicylate was not clearly demonstrated. She had a full range of motion. The Neer impingement sign was positive with a painful arc syndrome between 90” and 120”. Moreover, the palm-up test was also positive. Some “cracking” could be provoked during motion. The Constant score was 67, or 74% of what was expected foi the patient’s sex, age, and dominant side.3 The score for daily living activities was 13 of 20 possible ,points, and pain was 5 of 15 points. Elevation strength was measured isometrically at 90” of flexion at the level of the scapula with an electronic dvnamometer device (Kinedyne) and
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reached 9 pounds versus 13 on the normal side. No inflammatory laboratory tests were abnormal. Plain x-ray evaluation revealed a centered glenohumeral joint and a lesser tuberosity geode lesion at the medial border of the bicipital groove (Figure 1, A). The homogenous calcification of the supraspinatus tendon, which had led a few months before to the initial diagnosis of calcified tendinitis, had disappeared. Computed tomography scan confirmed an abnormality in this area (Figure 1, B). The center of the nidus was not radiopaque. This impingement syndrome has been resistant to medical treatment for more than 6 months and associated with an atypical lucent lesion. Thus surgery was indicated. A superior approach by Neer incision was performed. The subacromial bursa and the rotator cuff were normal. A bony expansion medial to the reflection of the biceps tendon on the humeral head caused the medial wall of the bicipital groove and the superior insertion of the subscapularis tendon to be raised. The biceps tendon was slightly inflamed and thickened. The coracoacromial ligament was resected, and total resection of the lesion was performed including the peripheral bone sclerosis. Before closure was performed, the partially divided subscapularis tendon was fixed to the resection hole with a suture anchor. The lesion was grey-white,* firm, and
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Figure
1 Plain x-ray biclpltal groove with arthrography Bupital
film of case 1 narrow peripheral groove IS lnlected
A, lucent
lesion IS seen on lesser tuberoslv, dense layer. B, Computed tomography and shows lesion slightly elevating tendon
Surg.
I997
medial to scan with
CASE 2 A 42-year-old man tendinitis of the right
was treated for calcifying dominant shoulder. In 18
months he received two local steroid injections,
Figure moved.
2 Macroscopic Grey-white center
specimen that was was slightly calcified
totally
re-
partially calcified (Figure 2). Histologic examination revealed osteoid trabeculae in well-vascularized connective tissue and some osteoblast activity. One year later the Constant score was 80, 89% on the corrected scale. There was some residual pain, which was attributed to bicipital tendinitis.
which were unsuccessful. Pain was the major symptom, especially at night, and was well controlled by nonsteroid drugs, especially salicylates. Examination showed positive impingement tests as in the previous patient. Mobility was normal, 40 of 40 points in the Constant score. The Constant score was 85, 88% on the corrected scale. Pain was 5 of 15 points, daily living activities were 15 of 20 points, and strength was 25 pounds, identical to the other side. Plain x-ray evaluation showed a classic “ring sequestrum” appearance localized subperiosteally near the inferior border of the humeral head (Figure 3, A). The medial location led to a deltopectoral ap preach at surgery and to a coracoid process osteotomy. After a coracoacromial ligament resection was performed, the rotator cuff was seen to be intact. The lesion was found medially on the lower part of the humeral neck after elevation of the periosteum of the humeral metaphysis. The tumor was soft, red, “raspberry like,“* and 5 mm wide in the middle of a dense cortical border. The tetracycline fluorescence test with ul-
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films of case 2 A shows “rln sequestrum” Figpre 3 Preoperative and postoperattve x-ra aspect. B shows coracoid osteotomy and tts fina Y osteosynthesis, residual hole me f IaI to humeral collar Some calcifications remain in suprasplnatus tendon near greater tuberosity This was responsible for delay in diagnosis
traviolet light was positive on the resected sample after curettage. This test confirmed total resection during operation. The dense layer was not removed. On histologic evaluation the lesion showed rich, well-vascularized osteoid tissue with osteoblastic activity,26 as is seen in classical 00 (Figure 4). At 5 years of follow-up the patient has recovered fully, tiith a corrected Constant score of 100%.
DISCUSSIOIU Osteoid osteoma was classically described by Jaffe13 in 19$5; first publications were probably earlier. Heine’ la reported one case in 1927, located on a first digital phalanx. In 1930 there were two published cases that were probably osteoid osteamas, one by Hitzrot in a carpal scaphoid bone and one by Bergstrand.12 Male patiemts outnumber female patients in two thirds of the cases. Eighty percent of the patients are younger than 30 years of age, unlike our two cases.2, 6, ” Iin a report by Dahli& 82% of the patients were younger than 24 years of age. According to many authors 00 progresses. However, some authors have described spontaneous resorptiotG4; this might explain the low frequency after the age of 30 years. Pain seems’ to develop, being sporadic at the beginning, in reasing at night, and having a tent dency to becpme permanent. Diagnosis is often late,14 with an average delay of 1 to 2 years.6, 24 This surprisibg tumoral lesion has limited growth potential6 and never reaches more than 1.5 cm in
diameter.12 A larger tumor would indicate osteoblastoma. Beyond the clinical, radiologic, and histologic characteristics, other unusual aspects of this lesion have been described. The location is frequently misleading. 00 can be located on any bone but seems to be less frequent in the humerus. Table 1 summarizes a review of 803 cases,” 2, 6, 9-12 with only 59 humeral cases and 13 of these at the upper end of the bone. Most of them were not described in detail. Only one patient from the “club des dix” serieslo presented with shoulder involvement. In this series the proximity to the joint led to a delayed and mistaken diagnosis, reinforced in both cases by the presence of impingement syndrome symptoms and calcifying tendinitis. In these cases it is difficult to say whether the pain was initially due to the calcifying tendinitis or to the presence of the 00. However, in the first case pain had persisted for 2 years despite the disappearance of the calcification. Pain disappeared after surgical removal of the 00. The second case was more easily recognized because the 00 was at the inferior border of the humeral neck, away from the calcification and from the biceps tendon. In the literature proximity to a joint can lead to three types of misleading clinical pictures.20 (1) fseudoarticular symptoms, with arthralgia and painful restricted range of motion simulating gout, bacterial or rheumatologic arthritis, neurodystrophy,20 neurologic disease, or osteochondritis dissecans.2 (2) lntraarticular disease.
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,,,-_,, ,.,, - .,,., .,.-.,,,Figure tissue
4
Histologic
section
of case
2 showing
Table I Number
Series Byers’ Cohen2 DahW Ghelman9 Gouin’O Healey’ I Huvos’* Total
312 95 245 1 56 17 77 803
Humerus 24 3 18 1 2 1 10 59
of Cases sup. metaphysis 2 1 5 1 4 13
Synovitis can lead to a misdiagnosis of deep infection25 or rheumatoid disease.‘, l’s 19, 20, z2 Moreover, some authors have described progressive secondary joint destruction.4,23 In our first case 00 was close to the biceps tendon and narrowed the bicipital groove. During the operation the tendon appeared to be damaged, and some pain persisted in the postoperative period. (3) Sequelae are the third group of clinical symptoms associated with 00 located near joints. In such locations 00 may result in growth disturbances with closure of the epiphyseal plates.20, 23 Some cases of progressive scoliosis have been published.2, 2O Roentgen features of intraarticular 00 can be classical as in our second patient, who presented
osteoblostlc
octivlty
In center
of dense
connective
with a dense calcified center in the middle of a radiolucent nidus surrounded by a sclerotic reaction. This appearance is clear, but misleading radiologic signs are frequently described. In the 100 cases reported by the Mayo Clinic,26 75 had a classical roentgen profile for 00, 17 had anomalous images that were not clearly 00 as in our first case, and 8 cases were radiologically normal. Peripheral sclerosis can be absent.2, 6, ” Edeiken et al.,’ in a review of 55 cases, found 24 radiolucent centers with peripheral sclerosis and 18 radiopaque centers without dense borders. Mazabraud,‘* like Edeiken et al.,’ described a peripheral density that varied according to the location of the nidus. If the nidus is cortical, the density of the peripheral sclerosis can hide the radiolucent lesion. In these cases some diminished or late pain has been recognized.’ ’ If the lesion is in the cancellous bone, the dense peripheral layer can be absent, as when the lesion is subperiosteal. Radiologic findings can reflect the age of the lesion as the center progressively calcifies, giving the typical “ring sequestrum” appearance.2 When xray films are not definitive, the best imaging technique for correct diagnosis seems to be a computed tomography scan .6 Scintigraphy can give some false-negative results.8, l6 Perioperative localization is important. X-ray evaluation, fluorescence with tetracycline, and in-
Katz and Thomazeau
/. Shoulder Elbow Surg. Volume 6, Number 6
traoperative radionuclide localization have been extensively described9 and are effective techniques to avoid cases of secondary recurrence. Missing the hidden lesion is in fact the main cause of postoperative recurrence. In conclusion, although the classical appearance of 00 might seem to facilitate diagnosis, the two cases in this study, located close to the shoulder, once agclin show the difficulties of this particular manifestaltion of the disease. Osteoid osteoma can present with a wide variety of clinical, radiologic, and hisiologic features.
1
Byers PD SoIltory benign osteoblostlc osteomo and benign osteoblastomo 57
2
Cohen teomo Rheum
MD, Harnngton TM, 95 cases and o revfew 1983; 12 2658 1
Ginsburg WW of the literature
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Corbett artlcular 1974,98
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CORRECTION In the article, “Dissociation of modular humeral head components: A Qomechanical and implant retrieval study” (Blevins FT, Deng X, Torzilli PA, Qnes D, Warren RF.J Shoulder Elbow Surg 1997, 6: 1 13-24), all mentions of Nomet should be Bio-Modular. Bio-Modular is the brand name; Biomet is the cbmpany that makes the Bio-Modular brand.
In os-
osteoma